Complete hydatidiform mole with coexisting dichorionic diamniotic twins following testicular sperm extraction and intracytoplasmic sperm injection

We present the first report of complete hydatidiform mole (HM) with coexisting dichorionic diamniotic twins. This pregnancy was achieved after testicular sperm extraction and intracytoplasmic sperm injection (ICSI) for azoospermia in the woman's husband. Standard in vitro fertilization may cause multisperm fertilization and increase triploid partial HM and complete HM, which arise from dispermic fertilization. In contrast, ICSI can avoid multisperm fertilization. In our case, paternal isodisomy in the molar tissue was confirmed by microsatellite analysis suggesting that it resulted from duplication of a haploid paternal genome following monospermic fertilization of an inactivated oocyte or from monospermic fertilization of an inactivated oocyte with a diploid sperm. Although the patient was eager to continue the pregnancy, the size of the HM component increased rapidly and termination of the pregnancy was required for pre-eclampsia-like symptoms at 15 weeks of gestation. After the operation, chemotherapy was initiated for persistent trophoblastic disease.     

Genetically identified complete hydatidiform mole coexisting with a live twin fetus: comparison with conventional diagnosis

Twin pregnancy consisting of a complete hydatidiform mole (CHM) along with a live co-existing fetus is a rare entity and difficult to diagnose. A 37-year-old Japanese woman demonstrated a living fetus, a placenta and a multicystic mass within one gestational sac on ultrasound at 10 weeks. Termination of the pregnancy was performed, and the specimen was classified as partial mole by macro- and microscopic findings. The karyotype of the molar tissue was 46XX. DNA polymorphism analysis demonstrated that fetal DNA showed bi-parental origin while molar DNA showed paternal origin only. Thus, this case was erroneously classified by ultrasonography, macroscopic and pathologic findings, then correctly diagnosed as a twin pregnancy with a CHM and co-existing normal twin fetus by DNA polymorphism analysis. Immunohistochemistry of p57(KIP2), the paternally imprinted and maternally expressed gene, supported the genetic diagnosis. This case suggested that conventional diagnostic methods were inadequate for accurate diagnosis of CHM with a co-existing fetus. DNA polymorphism analysis should be requested for the diagnosis of hydatidiform mole, especially in cases where it is difficult to discriminate between partial hydatidiform mole and CHM with a co-existing fetus.     

Persistent trophoblast disease following partial molar pregnancy

OBJECTIVE: Human chorionic gonadotrophin (hCG) follow-up data were analysed retrospectively in all patients registered in the Hydatidiform Mole Registry at the Royal Women's Hospital, Melbourne from January 1992 to January 2001 to determine the risk of persistent trophoblast disease following partial molar pregnancy and to review the present follow-up protocol of patients suffering from partial hydatidiform molar pregnancy (PHM). METHODS: Demographic factors were determined for all 344 cases with a review diagnosis of PHM, included age, history of previous hydatidiform mole, gestation length, hCG levels and compliance with follow-up. FINDINGS: Six of the 344 patients diagnosed with PHM required treatment with single-agent methotrexate and folinic acid rescue. All six patients achieved and maintained a complete biochemical remission after chemotherapy. hCG regression assays were analysed for 235 patients: 225 patients had at least one normal hCG measurement during follow-up, of whom 152 (64.7%) patients obtained normal values within 2 months after evacuation. All patients obtained normal levels within 32 weeks after evacuation of the partial hydatidiform mole. Only 63 (25.6%) patients completed the recommended follow-up program. No patient who achieved normal hCG levels required chemotherapy because of a recurrent gestational trophoblastic tumour. RECOMMENDATIONS: This study indicates that 1.7% of all partial mole pregnancy patients needed treatment for malignant sequelae. In contrast, no patient diagnosed with partial mole had a biochemical or clinical relapse after achieving normal levels of hCG, consistent with previous studies. Patients who have had a partial hydatidiform mole should be followed by hCG assays until normal levels are achieved and then follow-up can be safely discontinued.     

Complete hydatidiform mole coexisting with a live fetus

The co-existence of a hydatidiform mole with a living fetus is a rare phenomenon. The condition is a dilemma with respect to the diagnosis and management of associated maternal (a risk of maternal complications, such as preeclampsia, hyperthyrodism, and a risk of malignancy) and fetal (elevated risk of spontaneous abortion, neonatal thyrotoxicosis) complications. A 27-year-old woman was referred to our hospital with a diagnosis of hydatidiform mole and live fetus. The pregnancy was unremarkable except for the complaints of excessive nausea and vomiting. Successive ultrasound examinations demonstrated a normally growing live fetus (14 weeks) alongside a normal placenta and an additional intrauterine echogenic mass with features of hydatidiform mole. Genetic amniocentesis at 18 weeks' of gestation showed normal diploid fetal karyotype. At 20 weeks of pregnancy, a control prenatal visit revealed intrauterine fetal death. The follow-up period for two years was unremarkable. In the case of a normal fetal karyotype and the absence of serious signs of maternal pathology, waiting until fetal viability is achieved can justifiably be proposed, however there is still a risk of prenatal complications such as intrauterine death.     

Complete hydatidiform mole coexisting with a live fetus after gamete intrafallopian transfer. A case report

BACKGROUND: Molar pregnancy with a coexisting live fetus is a rare occurrence. We report the only known case with a surviving coexistent fetus after gamete intrafallopian transfer (GIFT). CASE: After GIFT, a 28-year-old primary infertility patient was diagnosed as having a complete hydatidiform mole coexisting with a live fetus at 13 weeks of gestation. At 36 weeks of gestation, a cesarean section was performed due to elevated serum human chorionic gonadotropin (hCG) levels, and a male infant with a normal appearance and weighing 2,688 g was delivered. CONCLUSION: If the patient desires to try to carry the fetus to viability after counseling on the possible associated risks of malignancy, it is possible to achieve fetal viability if (1) there is decline in the serum hCG level after it peaks before the second trimester, (2) ultrasound reveals degeneration of the molar part, and (3) there are no complications of pregnancy.     

Complete hydatidiform mole and a coexistent viable fetus: report of two cases and review of the literature

OBJECTIVE: The aim of this study was to report the clinical features, management, and outcome of two cases of complete hydatidiform mole with a coexisting viable fetus and to review the literature. PATIENTS: In this article, we report on the well-documented follow-up of 2 cases of twin pregnancies with complete hydatidiform mole and a viable fetus, both of which ended with the delivery of a normal infant at 41 and 26 weeks of gestation. It is of interest that both pregnancies were achieved following induction of ovulation with hMG/hCG. Since 1977, the year in which complete and partial moles were characterized as distinct pathologic entities, 15 cases (including our 2) have been reported. RESULTS: Persistent GTT developed in eight patients (53.3%) and four patients (27.7%) developed metastatic disease. Seventy-five percent patients with persistent GTT were treated with single-agent chemotherapy. The median gestational age of the patients with subsequent persistent GTT was 34.5 weeks compared to 38 weeks in the patients without persistent GTT. CONCLUSION: Complete hydatidiform mole and coexistent fetus is a rare occurrence and is associated with an increased risk of persistent gestational trophoblastic tumor. Based on currently available information, it seems that in the presence of a stable pregnancy, normal karyotype, and a normal sonogram it is reasonable to allow the pregnancy to continue.     

Partial molar pregnancy and coexisting fetus with diploid karyotype

Objectives. To evaluate cases of partial hydatidiform mole coexisting with a live fetus, including an observation of our own, and to discuss the proper antenatal management of women wishing to continue with a partial molar pregnancy.

Method. A PubMed search was then undertaken, extending over the time period from 1975 to 2006, using the keywords ‘partial hydratidiform mole’, ‘hydatidiform mole’ and ‘coexisting fetus’.

Results. At 16 weeks of gestation, an ultrasonographic examination revealed a normal fetus with an extremely large, multicystic placenta. The woman was informed of future risks but wished to continue with the pregnancy. The pregnancy progressed until 28 weeks without any complication but ended spontaneously with a vaginal delivery; the fetus had died in utero. Pathologic examination of the placenta revealed areas of hydropic degeneration and necrosis. Including our own observation, 17 cases of partial hydatidiform mole associated with a fetus of normal karyotype have been documented.

Conclusion. Although the rate of adverse perinatal outcome is high, we still believe that if amniocentesis or fetal blood sampling reveals a normal karyotype, then continuing the affected pregnancy with close follow-up in tertiary centers is a feasible choice.     

Radioimmunoassay of maternal serum -fetoprotein associated with pregnancy

Alpha fetoprotein (AFP) was measured by radioimmunoassay of urine and serum of pregnant women at various stages of gestation and postpartum; sera of patients with hydatidiform mole and choriocarcinoma were also measured. Concentrations below 5 ng/ml were considered negative. Nonpregnant and early pregnancy levels of AFP did not differ significantly. At 14 weeks of gestation, serum AFP showed an initial increase which increased progressively throughout pregnancy, reaching its maximum 35-38 weeks postconception. Average serum AFP was 232.6 ng/ml at 35 weeks, 253.3 at 36 weeks, 222.3 at 37 weeks, and 185.7 at 38 weeks. At term, serum AFP leveled off slightly, averaging 108.7 ng/ml at 40 weeks, 86.5 at 41 weeks, and 80.8 at 42 weeks. Serum AFP decreased rapidly after delivery and was almost negligible by 20 days postpartum. Average AFP half-life was 3.9 days. AFP was also detected in serum from 1 patient with hydatidiform mole (105 ng). AFP was found in urine of 1 patient with marked albuminuria but not in normal pregnancy. Results indicated that AFP in serum may be partly of maternal origin. It is speculated that the inhibition against the repressor-operator gene system which was responsible for AFP synthesis was accelerated in pregnancy by unknown factors, probably of placental origin, resulting from maternal AFP production.     

Lutein cysts in normal twin pregnancy leading to erroneous diagnosis of hydatid mole; a case report

A report is presented of a case of twin pregnancy in which there were many signs and symptoms and clinical laboratory findings commonly associated with the presence of either a hydatidiform mole or a chorionepithelioma, but there was no evidence found in the uterus or its contents of either of these two diseases.     

First-trimester molecular diagnosis of complete hydatidiform mole associated with dizygotic twin pregnancy conceived by intrauterine insemination

ObjectiveTo present first-trimester molecular diagnosis of complete hydatidiform mole (CHM) associated with dizygotic twin pregnancy conceived by intrauterine insemination.Materials and methodsA 32-year-old woman presented to the hospital with a huge complex cystic mass measuring about 8.5 cm × 4.1 cm in the uterine cavity and a living co-existing fetus with fetal biometry equivalent to 9 weeks. She underwent chorionic villus sampling at 13 weeks of gestation, and microsatellite genotyping for molar pregnancy test was applied. A molar pregnancy test was performed by a short tandem repeat (STR) identifier polymerase chain reaction (PCR) polymorphic marker analysis. The pregnancy was terminated at 14 weeks of gestation. Postnatal polymorphic DNA marker analysis of the placenta by quantitative fluorescent PCR (QF-PCR) was performed. Analysis of maternal blood total β-human chorionic gonadotropin revealed a high level of 551,600 mIU/mL at 10 weeks of gestation and a level of 1.0 mIU/mL at 15 weeks postpartum. The woman was doing well at 4 months after delivery.ResultsThe results of STR identifier PCR polymorphic marker analysis showed androgenic conception in the complex cystic mass and biparental conception in the living fetus. Pathological analysis of the cystic mass confirmed the diagnosis of CHM. The results of QF-PCR showed biparental inheritance in the normal fetus and complete paternal homozygosity in the CHM of the abnormal fetus in all STRs, indicating dizygotic twinning and CHM of monospermy.ConclusionPrenatal sonographic diagnosis of placentomegaly with many grape-like vesicles should include a differential diagnosis of CHM, partial hydatidiform mole (PHM), placental mesenchymal dysplasia (PMD), and recurrent hydatidiform mole. Microsatellite genotyping for molar pregnancy testing and zygosity testing is useful in cases of prenatal diagnosis of placentomegaly associated with many grape-like vesicles and a twin pregnancy with a living fetus in the first trimester.     

Partial molar pregnancy and coexisting fetus with diploid karyotype.

OBJECTIVES: To evaluate cases of partial hydatidiform mole coexisting with a live fetus, including an observation of our own, and to discuss the proper antenatal management of women wishing to continue with a partial molar pregnancy. METHOD: A PubMed search was then undertaken, extending over the time period from 1975 to 2006, using the keywords 'partial hydratidiform mole', 'hydatidiform mole' and 'coexisting fetus'. RESULTS: At 16 weeks of gestation, an ultrasonographic examination revealed a normal fetus with an extremely large, multicystic placenta. The woman was informed of future risks but wished to continue with the pregnancy. The pregnancy progressed until 28 weeks without any complication but ended spontaneously with a vaginal delivery; the fetus had died in utero. Pathologic examination of the placenta revealed areas of hydropic degeneration and necrosis. Including our own observation, 17 cases of partial hydatidiform mole associated with a fetus of normal karyotype have been documented. CONCLUSION: Although the rate of adverse perinatal outcome is high, we still believe that if amniocentesis or fetal blood sampling reveals a normal karyotype, then continuing the affected pregnancy with close follow-up in tertiary centers is a feasible choice.     

Repetitive complete and partial hydatidiform mole

Fifteen patients with repetitive hydatidiform mole were followed at the New England Trophoblastic Disease Center between 1965-1988. The medical records were examined to determine the patients' age, gravidity, parity, clinical presentation, development of post-molar tumor, and subsequent pregnancy experience. Each molar tissue was reviewed pathologically. Seven patients had repetitive complete hydatidiform mole and three developed persistent post-molar disease after their later mole. Five patients had an initial complete hydatidiform mole followed by a partial hydatidiform mole; two developed persistent post-molar disease after the partial mole. One patient had an initial partial hydatidiform mole followed by a complete hydatidiform mole and required chemotherapy after her complete mole. Two patients had repetitive partial hydatidiform mole, and neither developed post-molar disease. Four of the patients with repetitive mole later achieved a normal viable pregnancy. Molar pregnancies must be categorized as either complete or partial to provide meaningful data concerning repetitive hydatidiform mole.     

Hydatidiform mole in a triplet pregnancy following gonadotropin therapy

A first case is reported of complete hydatidiform mole with two coexistent fetuses in a triple pregnancy following human menopausal gonadotropin human chorionic gonadotropin (hMG-hCG) therapy. The molar mass and two fetuses were delivered separately at 17 weeks of gestation. The fetuses were female (155 g) and male (160 g) with individual placentae (85 g, 90 g). The hydatidiform mole (650 g) had a normal 46,XX karyotype. The sexes of the two fetuses and the karyotype of the mole are consistent with previous reports that the chromosomes of fetuses and moles are derived from both parents and the father, respectively.     

Gestational trophoblastic tumor in pregnancy: a case report and review of the literature

A case of gestational trophoblastic tumor (GTT) concurrent with an intrauterine pregnancy is reported in a 21-year-old gravida 2 para 0 (0010) who presented with dyspnea, orthopnea, headache, and blurring of vision at 33 weeks age of gestation. She had a history of hydatidiform mole for which curettage was done. Chest radiograph showed pulmonary metastases, with pleural effusion on both lungs. Serum beta subunit of human chorionic gonadotropin was abnormally elevated for age of gestation. Due to worsening maternal pulmonary condition, the patient underwent primary, low segment cesarean section and was subsequently started on multidrug chemotherapy. This is the first reported case of GTT in pregnancy in the Philippines in which both the mother and the infant survived. Several hypotheses regarding its origin, its diagnosis, and its management and prognosis are presented. This case report emphasizes the importance of early diagnosis and treatment to improve the prognosis of both the mother and the infant.     

Epidemiology of hydatidiform mole during early gestation

Different methodologic approaches have resulted in a wide variability in the reported incidence of hydatidiform mole in the United States. We studied the epidemiology of hydatidiform mole among 84,318 women obtaining legal abortions in the United States during the period 1975 to 1978. Our estimated rate of 7.5 hydatidiform moles per 10,000 pregnancies was similar to most reported rates for the United States. The rate was significantly higher for women 14 years or younger and those 45 years or older. Race, number of previous pregnancies, and number of previous spontaneous abortions were not found to significantly affect the risk of having the disease. The incidence was slightly (but not significantly) higher for women obtaining abortions during the first trimester than for those obtaining abortions during later gestation. This study minimizes the errors encountered in estimating the total number of pregnancies and the number of hydatidiform moles at later stages of gestation. Hydatidiform mole, although relatively the number of hydatidiform moles at later stages of gestation. Hydatidiform mole, although relatively uncommon, represents a complication of pregnancy that is of public health importance.     

双胎之一完全性葡萄胎的产前诊断及处理

目的 探讨双胎之一完全性葡萄胎(a twin pregnancy consisting of acomplete mole and coexisting fetus,CMCF)的产前诊断及处理。方法 回顾性分析2例CMCF的临床资料。结果 第1例患者在孕10周时,B超发现胎儿与葡萄胎胎盘共存,患者要求终止妊娠,刮宫物间期细胞荧光原位杂交(fluorescent in situ hybridization,FISH)和核型分析提示,胎儿与葡萄胎均为二倍体,证实为CMCF。第2例患者在孕21周时,B超发现胎儿与葡萄胎胎盘共存,B超引导下经腹壁绒毛活组织检查(活检)和羊膜腔穿刺,葡萄胎和羊水的间期细胞FISH和核型分析提示,胎儿与葡萄胎均为二倍体,证实为CMCF双胎之一完全性葡萄胎,患者继续妊娠,在孕28周时胎膜早破,因继发感染而行剖宫产终止妊娠,胎儿存活,胎盘、新生儿外周血的核型分析结果与产前诊断相符。结论 产前一旦发现胎儿与葡萄胎胎盘共存,应立即进行CMCF和部分性葡萄胎(partial hydatidiform mole,PHM)的鉴别,如果绒毛和羊水行间期细胞FISH和核型分析为二倍体,则为CMCF,是否继续妊娠,需采取个体化原则;如果为三倍体,则为部分性葡萄胎,应及时终止妊娠。     

Video Hysteroscopy in the Diagnosis of Molar Pregnancy in two Challenging Situations: Complete Mole with Normal hCG and Partial Mole with Early Gestational Age

Study ObjectiveTo present the first hysteroscopic findings of 2 cases of complete hydatidiform mole (CHM) and partial hydatidiform mole (PHM) within the context of the patients’ clinical histories.DesignPresentation of 2 hysteroscopic videos with narration of the intrauterine findings of molar pregnancy (MP) from Rio de Janeiro Gestational Trophoblastic Disease Reference Center.SettingMP is characterized by abnormal fertilization that generates 2 clinical syndromes: CHM and PHM [1].InterventionsIn the first case, the patient was aged 50 years, and hysteroscopy was indicated to assess abnormal uterine bleeding in the presence of normal serum human chorionic gonadotropin (hCG) and transvaginal ultrassonography showing an endometrial cavity with heterogeneous content. Hysteroscopy found translucent hydropic structures diagnosed as CHM. The negative hCG value was due to the hook effect (hCG after dilution: 2 240 000 IU/L). In the second case, an 18-year-old patient underwent hysteroscopy to assess the endometrial cavity with retained abortion at 7 weeks in which, during conservative management, the hCG level increased over 4 weeks from 25 000 IU/L to 58 000 IU/L. Hysteroscopy visualized the embryo with its umbilical cord and hydatidiform vesicles diagnosed as PHM.ConclusionMP can be an incidental finding during hysteroscopy for abnormal uterine bleeding or retained abortion 2, 3, 4. Knowing its morphology during hysteroscopy is helpful for the correct management of this uncommon clinical situation. Hysteroscopy as an adjunct diagnostic tool (not as first-line treatment for MP) can be of significant benefit in challenging clinical scenarios. Further studies should assess the possible risk of spreading molar cells into the peritoneal cavity owing to hysteroscopic fluid.     

Ptyalism and Partial Hydatidiform Mole Associated with a Normal Term Male Fetus

The case is described of a subfertile woman whose only successful pregnancy was complicated by the unusual combination of ptyalism and a partial hydatidiform mole. Review after delivery of an ultrasonic scan taken at 17 weeks showed the mole to be present at that time. A normal male child was delivered at term. It is concluded that there was no causal relationship between these 2 conditions.     

Twin pregnancy with a complete hydatidiform mole and co-existent live fetus: two case reports and review of the literature

Introduction  The aim of this study was to report the clinical features, management, and outcome of two cases of complete hydatidiform mole with a coexisting viable fetus and to review the literature. Case reports  In this article, we report on the well-documented follow-up of two cases of twin pregnancies with complete hydatidiform mole and a normal fetus. Genetic amniocentesis showed normal fetal karyotype in both of two cases. In the first case, a live male infant was delivered by a cesarean section because of severe maternal bleeding at 29 weeks of gestation. In the second case, termination of pregnancy was performed due to early onset of severe preeclampsia and vaginal hemorrhage. Conclusion  The chances of a live birth have been estimated between 30 and 35% and the risk of persistent trophoblastic disease is similar to singleton molar pregnancies in complete mole with coexisting fetus pregnancy. Therefore, in these pregnancies, expectant management instead of termination of pregnancy can be suggested.